Swallowing Symptoms in Female FMR1 Premutation Carriers: Eating Assessment Tool-10 Findings and Associations With Genetic and FXTAS–Related Features

Purpose: The fragile X messenger ribonucleoprotein 1 ( FMR1 ) premutation (FXpm) is a prevalent genetic variant that confers risk for neurodegenerative motor impairments associated with fragile X–associated tremor/ataxia syndrome (FXTAS). Swallowing difficulties may be more common in FXpm carriers but remain understudied. This study investigates swallowing symptoms in FXpm women and their associations with FXTAS-related motor symptoms and FMR1 -related genetic variation. Method: Participants (85 FXpm women, 73 controls) completed the Eating Assessment Tool–10 and the FXTAS Rating Scale–Revised. Genetic measures included cytosine-guanine-guanine repeat length, activation ratio, and repeat size mosaicism. Hurdle models assessed group differences in symptom endorsement and severity. Item-level differences were examined, along with associations between swallowing symptom endorsement, motor scores, and genetic measures using logistic regression and exploratory correlations. Results: FXpm women were more likely to endorse swallowing symptoms than controls, particularly at lower education levels ( OR = 0.68, 95% confidence interval [CI; 0.49, 0.93], p = .017). Effort swallowing solids and food sticking in the throat were the strongest item-level differentiators of the groups. Parkinsonian motor features ( OR = 1.29, 95% CI [1.04, 1.59], p = .022) and premutation/full mutation (PM/FM) mosaicism ( OR = 7.72, 95% CI [1.12, 53.30], p = .038) were associated with higher likelihood of symptom endorsement. Conclusions: Findings suggest that some FXpm women, particularly those with lower educational attainment and PM/FM mosaicism, experience elevated swallowing symptom burden, which may reflect early manifestations of motor system involvement. Swallowing symptoms warrant greater clinical attention in this population, with further research integrating comprehensive swallowing assessments needed to clarify the nature, progression, and clinical significance of swallowing symptoms in FXpm women. Supplemental Material: https://doi.org/10.23641/asha.34750941

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Journal
American Journal of Speech-Language Pathology
Published
2026-10-26
DOI
https://doi.org/10.1044/2026_ajslp-26-00166
Primary Topic
Dysphagia Assessment and Management
Type
article
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article

Swallowing Symptoms in Female FMR1 Premutation Carriers: Eating Assessment Tool-10 Findings and Associations With Genetic and FXTAS–Related Features

Heather Shaw Bonilha, Elizabeth Berry‐Kravis, Amanda Jane Fairchild, Michelle Hyczy de Siqueira Tosin et al.
American Journal of Speech-Language Pathology
Dysphagia Assessment and Management
article

Swallowing Symptoms in Female FMR1 Premutation Carriers: Eating Assessment Tool-10 Findings and Associations With Genetic and FXTAS–Related Features

Heather Shaw Bonilha, Elizabeth Berry‐Kravis, Amanda Jane Fairchild, Michelle Hyczy de Siqueira Tosin, Thomas R. Christensen, Jessica Klusek, Federico E Rodriguez‐Porcel, Deborah A. Hall, Christine A. Cooper, Sarah Reviel
article en

Abstract

Purpose: The fragile X messenger ribonucleoprotein 1 ( FMR1 ) premutation (FXpm) is a prevalent genetic variant that confers risk for neurodegenerative motor impairments associated with fragile X–associated tremor/ataxia syndrome (FXTAS). Swallowing difficulties may be more common in FXpm carriers but remain understudied. This study investigates swallowing symptoms in FXpm women and their associations with FXTAS-related motor symptoms and FMR1 -related genetic variation. Method: Participants (85 FXpm women, 73 controls) completed the Eating Assessment Tool–10 and the FXTAS Rating Scale–Revised. Genetic measures included cytosine-guanine-guanine repeat length, activation ratio, and repeat size mosaicism. Hurdle models assessed group differences in symptom endorsement and severity. Item-level differences were examined, along with associations between swallowing symptom endorsement, motor scores, and genetic measures using logistic regression and exploratory correlations. Results: FXpm women were more likely to endorse swallowing symptoms than controls, particularly at lower education levels ( OR = 0.68, 95% confidence interval [CI; 0.49, 0.93], p = .017). Effort swallowing solids and food sticking in the throat were the strongest item-level differentiators of the groups. Parkinsonian motor features ( OR = 1.29, 95% CI [1.04, 1.59], p = .022) and premutation/full mutation (PM/FM) mosaicism ( OR = 7.72, 95% CI [1.12, 53.30], p = .038) were associated with higher likelihood of symptom endorsement. Conclusions: Findings suggest that some FXpm women, particularly those with lower educational attainment and PM/FM mosaicism, experience elevated swallowing symptom burden, which may reflect early manifestations of motor system involvement. Swallowing symptoms warrant greater clinical attention in this population, with further research integrating comprehensive swallowing assessments needed to clarify the nature, progression, and clinical significance of swallowing symptoms in FXpm women. Supplemental Material: https://doi.org/10.23641/asha.34750941

American Journal of Speech-Language Pathology
Rush University Medical Center (US), Medical University of South Carolina (US), University of South Carolina (US), Department of Health Services (US)
National Institute on Aging
Openalex Percentile: Top 7%
Dysphagia Assessment and Management
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